Spontaneous spleen rupture and rectus sheath hematoma in a patient with Klippel-Trenaunay syndrome: report of a case

Surg Today. 2010;40(2):154-7. doi: 10.1007/s00595-008-4008-z. Epub 2010 Jan 28.

Abstract

We report a case of Klippel-Trenaunay syndrome (KTS) with serious morbidity caused by the rupture of hemangiomas of the spleen and inferior epigastric artery (IEA). A 40-year-old woman, who had suffered from edema and varicose veins in her left leg and toes since birth, underwent emergency laparotomy and splenectomy for a spontaneous splenic rupture. Pathological examination revealed hemangiomatosis of the spleen. She presented again 40 days later with a rectus muscle hematoma, which computed tomography revealed to be actively bleeding. Arteriography confirmed a bleeding IEA, which was then embolized. Hematological investigation revealed a heterozygous form of factor VIII and fibrinogen deficiency. The patient recovered well and was asymptomatic at her 1-year follow-up. We report this case to reinforce that investigations for KTS should involve all organ systems, and include detailed hematologic tests. By defining coagulation and vascular abnormalities, life-threatening bleeding episodes may be prevented.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • Epigastric Arteries / diagnostic imaging
  • Epigastric Arteries / pathology
  • Female
  • Hemangioma / diagnosis
  • Hemangioma / etiology*
  • Hematoma / diagnosis
  • Hematoma / etiology*
  • Humans
  • Klippel-Trenaunay-Weber Syndrome / complications*
  • Radiography
  • Rectus Abdominis / diagnostic imaging
  • Rupture, Spontaneous
  • Splenic Rupture / diagnosis
  • Splenic Rupture / etiology*
  • Treatment Outcome